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Báo cáo khoa học: "Post-traumatic soft tissue tumors: Case report and review of the literature a propos a Post-traumatic paraspinal desmoid tumor"
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- World Journal of Surgical Oncology BioMed Central Open Access Case report Post-traumatic soft tissue tumors: Case report and review of the literature a propos a Post-traumatic paraspinal desmoid tumor Sarit Cohen1, Dean Ad-El1, Ofer Benjaminov2 and Haim Gutman*3 Address: 1Department of Plastic Surgery, Rabin Medical Center, Beilinson Campus, Petah Tiqwa; and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel, 2Department of Diagnostic Imaging, Rabin Medical Center, Beilinson Campus, Petah Tiqwa; and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel and 3Department of Surgery, Rabin Medical Center, Beilinson Campus, Petah Tiqwa; and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel Email: Sarit Cohen - sariti@zahav.net.il; Dean Ad-El - deana@clalit.org.il; Ofer Benjaminov - obenjami@netvision.net.il; Haim Gutman* - hgutman@post.tau.ac.il * Corresponding author Published: 29 February 2008 Received: 19 June 2007 Accepted: 29 February 2008 World Journal of Surgical Oncology 2008, 6:28 doi:10.1186/1477-7819-6-28 This article is available from: http://www.wjso.com/content/6/1/28 © 2008 Cohen et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Abstract Background: Antecedent trauma has been implicated in the causation of soft tissue tumors. Several criteria have been established to define a cause-and-effect relationship. We postulate possible mechanisms in the genesis of soft tissue tumors following antecedent traumatic injury. Case presentation: We present a 27-year-old woman with a paraspinal desmoid tumor, diagnosed 3-years following a motor vehicle accident. Literature is reviewed. Conclusion: Soft tissue tumors arising at the site of previous trauma may be desmoids, pseudolipomas or rarely, other soft tissue growths. The cause-and-effect issue of desmoid or other soft tissue tumors goes beyond their diagnosis and treatment. Surgeons should be acquainted with this diagnostic entity as it may also involve questions of longer follow-up and compensation and disability privileges. Background Case presentation The etiology of most soft tissue tumors is unknown. Our A 27-year-old woman presented with a large subcutaneous search of the English literature revealed a few case reports mass in the upper back (Figure 1) of 8 months' duration. of soft tissue tumors developing at the site of a previous traumatic injury [1-17]. Desmoid tumors, lipoma and Family history and past medical history were unremarka- lymphoma were among the tumors reportedly associated ble. The patient reported that she had been involved in a with such injuries. motor vehicle accident 3 years previously, in which she sustained a brain concussion, fracture of the right lamina We describe a young woman with a left paraspinal of the C-6 vertebra, and comminuted fractures of the left desmoid tumor at the site of a recent trauma, possibly radius, ulna and femur. associated with a cause-and-effect mechanism. We hope this study will shed more light on this phenomenon. Physical examination revealed a firm mass measuring 15 × 10 cm, adherent to its surroundings, with no apparent pathological vasculature or satellite lesions. Cytological Page 1 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:28 http://www.wjso.com/content/6/1/28 rial, demonstrating its vascularity. Findings on core needle biopsy were compatible with desmoid tumor. Colonos- copy revealed no abnormalities. Owing to the large size of the tumor and its close proxim- ity to the spine, the initial treatment consisted of tamoxifen 20 mg twice daily and indomethacin 250 mg q8h. The treatment was well tolerated. However, after 4 months, neither subjective nor objective changes in tumor consistency or size were noted. The tamoxifen dosage was therefore doubled. Computerized tomography (CT) scan, 4 months later demonstrated tumor growth. There was no evidence of infiltration of adjacent bony structures or pul- monary metastases. The patient was offered surgery. The tumor was surgically excised. It measured 9 × 12 × 22 cm and weighed 1970 grams. It was relatively well circum- Figure 1 Large subcutaneous mass in the left paraspinal region scribed, with a fibrous consistency, and no areas of hem- Large subcutaneous mass in the left paraspinal region. orrhage or necrosis. Microscopic study revealed relatively low (up to 2–3/10HPF) mitotic activity (Figure 3, 4). The surgical margins were clear. At present, 24 months post- operatively, the patient is tumor-free. Discussion Desmoid tumor is a benign, locally aggressive neoplasm that arises from fascial or musculoaponeurotic tissue. It has a tendency to infiltrate surrounding tissue. The term 'desmoid', derived from the Greek "desmos" which means tendon-like was first employed by Müller [12] in 1838. Desmoid tumors account for 0.03% of all neoplasms [13,14], and 3.0% of all soft tissue tumors [15,16]. Patients with familial adenomatous polyposis (FAP) have a 1000-fold increased risk of developing desmoid tumors Figure 2 (D) injection, T2W (C) and pre-(A) and post-(B) sagittal view MRI of the tumor: T1WT1W post gadolinium,gadolinium MRI of the tumor: T1W pre-(A) and post-(B) gadolinium injection, T2W (C) and T1W post gadolinium, sagittal view (D). The tumor (arrows) has a heterogenous appearance on T2W images and enhances with the injection of contrast material, demonstrating its vascularity. It is located beneath the trapezius muscle (asterisk) which is atrophic. The parasp- inal muscle is compressed medially. examination was inconclusive. Magnetic resonance imag- ing (MRI) demonstrated a solid space-occupying lesion measuring 12 × 4.8 × 7.6 cm, located in the left paraspinal region beneath the trapezium muscle (asterisk), com- Figure 3 ation without significant atypia or pleomorphism (HE × 40) Histopathologic specimen demonstrating spindle cell prolifer- pressing the paraspinal muscles medially (Figure 2). The Histopathologic specimen demonstrating spindle cell prolifer- tumor has a heterogeneous appearance on T2 weighted ation without significant atypia or pleomorphism (HE × 40). images and enhanced with the injection of contrast mate- Page 2 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:28 http://www.wjso.com/content/6/1/28 desmoid following placement of an internal jugular cath- eter, and Wiel Marin et al., [2] described a thoracic desmoid tumor at the site of a previous rib fracture. Traumatic injury has been implicated as a causative factor in the genesis of other soft tissues as well. Radhi et al., [6] reported 3 cases of diffuse centroblastic lymphoma at a site of previous surgery with metallic implants. Two of them were preceded by atypical lymphoid infiltrate. In 1969, Brooke and MacGregor [21] suggested that lipoma may be secondary to trauma because of the pro- lapse of normal deep adipose tissue through a tear in the overlying Scarpa's fascia, namely, "pseudolipoma". Pseu- dolipoma consists of normal adipose tissue in an abnor- mal location, and is not considered a true lipoma because it is not encapsulated. Meggit and Wilson [22] reported 12 Figure 4 Photomicrograph at high power magnification (HE × 100) cases of post-traumatic so-called lipoma. They speculated Photomicrograph at high power magnification (HE × 100). that the tumors were the consequence of a rupture in the septa that normally surround adipose tissue. A later report compared to the general population. The abdomen is the by Herbert and DeGeus [23] described a young girl with most common site of the tumors in this patient group, an abdominal wall lipoma due to pressure from tightly fit- many times following a surgical insult. ting briefs. They demonstrated an anatomical defect in the Scarpa's fascia at the level of a perforating vessel with fat The reported female: male ratio for sporadic desmoid herniating through it. tumors is 5:2 [17]; most women are affected during or after pregnancy. Reitamo et al., [13] found that 80% of The largest series of 24 pseudolipomas was reported by desmoid tumors occur in females, 50% of them in the Rozner and Isaacs [24] in 1977, wherein scar contracture third to fifth decade of life. The female predominance is following a shearing fascial injury was the etiological less prominent in patients with FAP [18,19]. mechanism. Penoff [25] described 3 cases of traumatic lipoma of the hip, although he found no anatomic confir- Recently, It was found that virtually all desmoid tumors mation of an injury to Scarpa's fascia. have somatic [beta]-catenin or adenomatous polyposis coli (APC) gene mutation leading to intranuclear accu- In 1988, Dodenhoff [26] described a "saddle-bag deform- mulation of [beta]-catenin [20]. The expression of ity" of the right hip secondary to trauma. Post-traumatic nuclear [beta]-catenin may play a role in the differential lipoma was also reported by Elsahy [27] (5 cases) and diagnosis of desmoid tumors from a host of fibroblastic David et al., [8] (10 cases). Signorini and Campiglio [9] and myofibroblastic lesions as well as from smooth mus- described 9 cases of subcutaneous lipoma that appeared cle neoplasms [20]. The treatment of desmoid tumors is within a few months of a blunt trauma. They proposed usually surgical. Local recurrences may occur even after that the differentiation of mesenchymal precursors clear margin resection. Distant metastases are extremely (preadipocytes) to mature adipocytes – a process triggered rare. by the trauma – could lead to the formation of subcutane- ous lipoma. The pathogenesis of desmoid tumor may involve genetic abnormalities, sex hormones, and trauma [17], includ- Warren [28] listed several criteria defining a post-trau- ing surgical trauma, especially in patients with FAP [19]. matic neoplasm: (a) prior integrity of the tumor site; (b) One study found that 10–30% of all sporadic abdominal injury severe enough to initiate reparative proliferation of wall desmoid tumors occurred following surgical inter- cells; (c) reasonable latent period; and (d) tumor compat- vention. Half these tumors developed within 4 years of ible with the scar tissue and anatomic location of the surgery [17]. injury. Ewing [29] suggested slightly different criteria to establish a cause/effect relationship: (a) authenticity and Gebhart et al., [3] reported a case of desmoid tumor aris- severity of the injury; (b) previous integrity of the ing at the site of a total hip replacement. Desmoid tumors wounded part; (c) tumor originating within the boundary developing around silicone implants have also been of the injury; (d) histologic variety of tumor compatible described [13]. Skhiri et al., [1] reported a case of cervical with underlying scar tissue; and (e) proper latent period. Page 3 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:28 http://www.wjso.com/content/6/1/28 In our case, the wounded part (upper back) was previ- 2. Wiel Marin A, Romagnoli A, Carlucci I, Veneziani A, Mercuri M, Des- tito C: Thoracic desmoid tumors: a rare evolution of rib frac- ously tumor-free, the authenticity of the trauma was con- ture. Etiopathogenesis and therapeutic considerations. G firmed by MRI, the tumor originated within the boundary Chir 1995, 16:341-344. 3. Gebhart M, Fourmarier M, Heymans O, Alexiou J, Yengue P, De Saint- of the injury, and the latency period was reasonable. Fur- Aubain N: Development of a desmoid tumor at the site of a thermore, the desmoid histology was compatible with a total hip replacement. Acta Orthop Belg 1999, 65:230-234. scar or other reparative process. Thus, the tumor met the 4. Pereyo NG, Heimer WL 2: Extraabdominal desmoid tumor. J Am Acad Dermatol 1996, 34(2 Pt 2):352-356. criteria of both Warren [28] and Ewing [29] for post-trau- 5. Flores RAR: Abdominal desmoid tumors and the surgeon. Rev matic neoplasm. Gastroenterol Mex 1995, 60:207-210. 6. Radhi JM, Ibrahiem K, al-Tweigeri T: Soft tissue malignant lym- phoma at sites of previous surgery. J Clin Pathol 1998, Conclusion 51:629-632. The cause-and-effect issue of desmoid or other soft tis- 7. Delpla PA, Rouge D, Durroux R, Rouquette I, Arbus L: Soft tissue tumors following traumatic injury: two observations of inter- sue tumors goes beyond their diagnosis and treatment. est for the medicolegal causality. Am J Forensic Med Pathol 1998, It may also involve questions of longer follow-up and 19:152-156. compensation and disability privileges. 8. David LR, DeFranzo A, Marks M, Argenta LC: Posttraumatic pseu- dolipoma. J Trauma 1996, 40:396-400. 9. Signorini M, Campiglio GL: Posttraumatic lipomas: where do Pseudolipomas are not real neoplasia, but they seem to they really come from? Plast Reconstr Surg 1998, 101:699-705. 10. Copcu E, Sivrioglu NS: Posttraumatic lipoma: analysis of 10 account for the reports of the so-called post-traumatic cases and explanation of possible mechanisms. Dermatol Surg lipomas. The post-injury local reparatory mechanisms 2003, 29:215-220. better explain the creation of desmoid tumors, which, in 11. Bashara ME, Jules KT, Potter GK: Dermatofibrosarcoma protu- berans: 4 years after local trauma. J Foot Surg 1992, 31:160-165. these rare cases, seem to have lost control of cell growth, 12. Müller J: Veber den Feinern Bau und die Formen der Krankhaftlichen giving rise to a soft tissue tumor. The rarity of desmoid Geschwulste Berlin: G Reimer; 1838:80. tumor, its specific biology, the well-documented associ- 13. Reitamo JJ, Hayry P, Nykyri E, Saxen E: The desmoid tumor. I. Incidence, sex-, age- and anatomical distribution in the Finn- ation between abdominal wall desmoids and preg- ish population. Am J Clin Pathol 1982, 77:665-673. nancy, and even the tendency of surgery to induce new 14. Suit HD: Radiation dose and response of desmoid tumors. Int J Radiat Oncol Biol Phys 1990, 19:225-227. desmoid tumors in patients with FAP support the 15. Taylor LJ: Musculoaponeurotic fibromatosis. A report of 28 notion that trauma/tissue injury is a likely cause of at cases and review of the literature. Clin Orthop Relat Res 1987, least, some of these tumors, including the one described 224:294-302. 16. Nuyttens JJ, Rust PF, Thomas CR Jr, Turrisi AT 3rd: Surgery versus here. radiation therapy for patients with aggressive fibromatosis or desmoid tumors: A comparative review of 22 articles. Cancer 2000, 88:1517-1523. Abbreviations 17. Kulaylat MN, Karakousis CP, Keaney CM, McCorvey D, Bem J, Abrus CT-computerized tomography; FAP-familial adenoma- JL Sr: Desmoid tumor: a pleomorphic lesion. Eur J Surg Oncol tous polyposis; MRI-magnetic resonance imaging 1999, 25:487-497. 18. Shields CJ, Winter DC, Kirwan WO, Redmond HP: Desmoid tumors. Eur J Surg Oncol 2001, 27:701-706. Competing interests 19. Gurbuz AK, Giardiello FM, Petersen GM, Krush AJ, Offerhaus GJ, The author(s) declare that they have no competing inter- Booker SV, Kerr MC, Hamilton SR: Desmoid tumors in familial adenomatous polyposis. Gut 1994, 35:377-381. ests. 20. Bhattacharya B, Dilworth HP, Iacobuzio-Donahue C, Ricci F, Weber K, Furlong MA, Fisher C, Montgomery E: Nuclear [beta]-catenin expression distinguishes deep fibromatosis from other Authors' contributions benign and malignant fibroblastic and myofibroblastic CS participated in drafting the manuscript, interpretation lesions. Am J Surg Pathol 2005, 29:653-659. of data and conceptual design, AD conceived the study 21. Brooke RI, MacGregor AJ: Traumatic pseudolipoma of the buc- cal mucosa. Oral Surg Oral Med Oral Pathol 1969, 28:223-225. and participated in drafting the manuscript, BO carried 22. Meggitt BF, Wilson JN: The battered buttock syndrome: fat out the imaging analysis and interpretation of data, GH fractures: a report on a group of traumatic lipomata. Br J Surg 1972, 59:165-169. carried out the surgical procedure, conceptual design, par- 23. Herbert DC, DeGeus J: Post-traumatic lipomas of the abdomi- ticipated in drafting the manuscript and revised it criti- nal wall. Br J Plast Surg 1975, 28:303-306. cally for important intellectual content. 24. Rozner L, Isaacs GW: The traumatic pseudolipoma. Aust N Z J Surg 1977, 47:779-782. 25. Penoff JH: Traumatic lipomas/pseudolipomas. J Trauma 1982, All authors read and approved the final manuscript. 22:63-65. 26. Dodenhoff TT: Trauma induced saddle-bag: case report. Lipo- plasty Newsletter 1988, 5:55-57. Acknowledgements 27. Elsahy NI: Post-traumatic fatty deformities. Eur J Plast Surg 1989, Written consent was obtained from the patient for publication of this case 12:208-211. report. 28. Warren S: Minimal criteria required to improve causation of traumatic or occupational neoplasms. Ann Surg 1943, 117:585. 29. Ewing J: Buckley lecture: Modern attitude toward traumatic References cancer. Arch Pathol 1935, 19:690. 1. Skhiri H, Zellama D, Ameur Frih M, Moussa A, Gmar Bouraoui S, Achour A, Ben Dhia N, Zakhama A, Elmay M: Desmoid cervical tumor following the placing of an internal jugular catheter. Presse Med 2004, 33:95-97. (French) Page 4 of 4 (page number not for citation purposes)
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